Effect of genetically caused excess of brain gamma-hydroxybutyric acid and GABA on sleep.
Sleep
; 28(4): 418-24, 2005 Apr.
Article
em En
| MEDLINE
| ID: mdl-16171286
ABSTRACT
BACKGROUND:
Exogenous gamma-hydroxybutyrate (GHB) increases slow-wave sleep and reduces daytime sleepiness and cataplexy in patients with primary narcolepsy.OBJECTIVE:
To examine nighttime sleep and daytime sleepiness in a 13-year-old girl homozygous for succinic semialdehyde dehydrogenase (SSADH) deficiency, a rare recessive metabolic disorder that disrupts the normal degradation of 4-aminobutyric acid (GABA), and leads to an accumulation of GHB and GABA within the brain.METHODS:
Sleep interview, nighttime polysomnography, Multiple Sleep Latency Tests, and continuous 24-hour in-lab recordings in the patient; overnight polysomnography in her recessive mother and in a 13-year-old female control.RESULTS:
During quiet wakefulness, background electroencephalographic activity was slow and composed of 7-Hz activity. Sleep stage 3/4 was slightly increased (28.1% of total sleep period, norms 15%-28%), and the daytime mean sleep latency was short in the patient (3 minutes 42 seconds, norms > 8 minutes). Stage 2 spindles were infrequent in the child (0.18/minute, norms 1.2-9.2/minute) and her mother (0.65/minute) but normal (4.6/minute) in the control. At the beginning of the second night, a tonic-clonic seizure occurred, followed by a dramatic increase in stage 3/4 sleep, that lasted 46.3 % of the total sleep period, double the normal value. The mother showed a reduced total sleep time and rapid eye movement sleep percentage.DISCUSSION:
This suggests that a chronic excess of GABA and GHB induces subtle sleep abnormalities, whereas increased slow-wave sleep evoked by a sudden event (here an epileptic seizure) may be caused by a supplementary increase in GABA and GHB.
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Coleções:
01-internacional
Base de dados:
MEDLINE
Assunto principal:
Sono
/
Oxibato de Sódio
/
Encéfalo
/
Succinato-Semialdeído Desidrogenase
/
Ácido gama-Aminobutírico
/
Distúrbios do Sono por Sonolência Excessiva
Limite:
Adolescent
/
Female
/
Humans
Idioma:
En
Ano de publicação:
2005
Tipo de documento:
Article